CASE REPORT |
https://doi.org/10.5005/jp-journals-10023-1245 |
Lobular Capillary Hemangioma at an Unusual Location: A Rare Occurrence
1-5Department of ENT-HNS, Command Hospital Airforce, Bengaluru, Karnataka, India
Corresponding Author: Sangineedi Deepthi, Department of ENT-HNS, Command Hospital Airforce, Bengaluru, Karnataka, India, Phone: +91 8331873799, e-mail: deepthi.sangineedi5@gmail.com
Received on: 08 May 2023; Accepted on: 07 June 2023; Published on: 30 June 2023
ABSTRACT
Aim: The goal of this study was to document a rare case of lobular capillary hemangioma of the hypopharynx to create awareness among practitioners.
Background: Lobular capillary hemangioma (LCH) is a benign, rapidly growing vascular proliferation that presents as a hemorrhagic, red to purple, sessile, or pedunculated mass. They are present most commonly on the cutaneous and mucosal surfaces, such as the gingiva, lips, fingers, and face. It occurs very rarely in the hypopharynx.
Case description: We present a case of lobular capillary hemangioma of the left pyriform fossa in a 50 year old female who underwent micro-laryngoscopic excision, with no follow-up complications or recurrence.
Conclusion: The present study describes LCH of the hypopharynx, which is an uncommon pathology in terms of the site of lesion in the practice of otolaryngology.
Clinical significance: This case study illustrates the importance of documentation of such a rare case of LCH to create awareness among practitioners as surgery in this location is challenging due to the risk of bleeding and airway management and demands surgical expertise.
How to cite this article: Deepthi S, Singh R, Kumar S, et al. Lobular Capillary Hemangioma at an Unusual Location: A Rare Occurrence. Int J Phonosurg Laryngol 2023;13(1):16-18.
Source of support: Nil
Conflict of interest: None
Patient consent statement: The author(s) have obtained written informed consent from the patient for publication of the case report details and related images.
Keywords: Capillary hemangioma, Granuloma, Histopathology, Hypopharynx, Posterior pharyngeal wall, Pyriform sinus.
INTRODUCTION
Hemangiomas are a type of benign tumor formed from blood vessels. Based on their histology, they can be of three different varieties. They include capillary, mixed cavernous, or a sclerosing variety which can go into fibrosis. Hemangiomas can occur commonly in head and neck pathologies. They are commonly on the areas like lips, cheeks, and tongue.1
Hartzell coined the term pyogenic granuloma in 1904s. Initially, they were known to be granulation tissue which occurred as a result of an infection. The term LCH was first used by Mills in the year 1980 because of the characteristic appearance of histopathology.2 LCH is a benign tumor. They grow rapidly with vascular proliferation, usually present as red to purplish masses, which may be sessile or pedunculated. They can be seen over cutaneous membrane or mucous membrane, and these tumors can occur in intravascular or subcutaneous tissue rarely.3 They occur commonly over lips, nose, oral cavity, and tongue, more commonly seen in young males in their 20s and females between 25 and 45 years of age. In children, these tumors are more commonly seen.4
Histopathology shows an aggregation of capillaries arranged in one or more lobules. There may be an associated slight inflammatory reaction. The outermost layer is composed of atrophic/hyperplastic epithelium. The epithelium can be ulcerated at times and can also show fibrinous exudates within it. The vessels are capillary-sized and show lobulated proliferation. There can be the presence of connective tissue septa, which may be myxoid or fibrous, in between the lobules.2
The pyriform sinus is an area away from the glottis, where tumors can grow for a long time without any clinical symptoms.5 We present a case of lobular capillary hemangioma (LCH) in the pyriform sinus, which is not commonly seen in this location, and its clinical presentation radiological and histopathological findings.
CASE DESCRIPTION
A female patient aged 50 years presented with difficulty in swallowing and foreign body sensation in the throat for 2 months. She had no history of previous neck surgery or intubation. There was no history of odynophagia or change of voice. The patient had no history of weight loss or loss of appetite.
Video laryngoscopy showed an exophytic pinkish, globular, pedunculated fleshy mass in the left pyriform sinus reaching the posterior pharyngeal wall (Fig. 1). Bilateral vocal cords were normal and mobile.
Fig. 1: Video laryngoscopy showing the growth in the left pyriform sinus (arrow)
Contrast-enhanced computer tomography (CECT) of the neck showed a well-defined avidly enhancing pedunculated mass of 1.5 x 1.6 cm arising from the left pyriform sinus on the axial view (Fig. 2). The patient underwent micro laryngoscopic excision of the mass (Fig. 3). Cystic vascular growth was seen arising from the left pyriform sinus. Bleeding was controlled with bipolar cautery. The mass measured 1.6 x 1.5 x 0.8 cm and was greyish brown in appearance.
Fig. 2: Contrast-enhanced computer tomography (CECT) neck axial section showing mass epicentered at left pyriform sinus reaching till the posterior pharyngeal wall (arrow)
Fig. 3: Tumor of 1.5 × 1.6 cm after excision
Histopathology showed underlying connecting tissue with multiple thin-walled blood vessels arranged in a lobular pattern of varying caliber separated by intervening fibrocollagenous septae, which are lined by endothelial cells (Fig. 4).
Fig. 4: Histopathology showing multiple thin-walled blood vessels arranged in a lobular pattern (arrows)
Her postoperative recovery was uneventful. The patient is on regular follow-up to date and shows no signs of residual lesion or recurrence.
DISCUSSION
The hypopharynx can be anatomically divided into three subsites primarily: the pyriform sinuses, the postcricoid area, and the posterior pharyngeal wall. The pyriform sinus is a silent area that lies outside the glottis, where the tumors can grow for a long time before the appearance of symptoms.5
Lobular capillary hemangiomas (LCH) are also known as pyogenic granulomas. They can present as a bleeding mass without pain, which is found adherent to the mucosa or cutaneous membranes of the upper airways.4
They can occur over the cutaneous surface of fingers, face, or over the mucosal surfaces, gingiva, and lips. Initially, they were described as granulation of tissue due in reaction to infections even though there were no signs of infection in the histological and microbiological examination. Later it was found the features were more of an angiomatous lesion, so the term was changed to “LCH.”2
The cause is unknown, but according to certain theories, they correlate to a previous injury, hormonal imbalance, history of viral infection, Bartonella infection, production of angiogenic factors, and cytogenetic clonal deletions.4 Trauma can be considered as a triggering factor, although only 7–23% of patients with LCH have reported a previous history of injury over the site of the lesion.6
The lesions occurring in the nasal and oral mucous membranes have an unusual predilection for females during the reproductive period.7
In LCH of the nasal cavity, CECT can help in identifying the site of origin and see the extent of the lesion. Lee et al. have shown that CT features of an LCH lesion in the nasal cavity consist of an intensely enhancing mass and an iso or hypoattenuating cap of variable thickness around.2 They have utilized imaging in diagnosing the case.
There is a therapeutic challenge in treating these lesions considering their location, functional impairments, size, and bleeding risk. The patient in this report was appropriately treated surgically with no follow-up complications or recurrence. The uncommon findings in our case were the age of presentation of the patient and an unusual location, that is, the hypopharynx. It is possible that such cases are rather underreported than rare in India; they need to be documented to create awareness among practitioners.
CONCLUSION
The present study describes LCH of the hypopharynx, which is an uncommon pathology in terms of the site of lesion in the practice of otolaryngology. There were no complications or recurrences after undergoing excision. The surgery in this location is challenging due to the risk of bleeding and airway management and demands surgical expertise.
COMPLIANCE WITH ETHICAL STANDARDS
Research does not involve human participants and/or animals.
ORCID
Sangineedi Deepthi https://orcid.org/0000-0001-8487-6525
REFERENCES
1. Rachappa MM, Triveni MN. Capillary hemangioma or pyogenic granuloma: a diagnostic dilemma. Contemp Clin Dent 2010;1(2):119–122. DOI: 10.4103/0976-237X.68593
2. An TH, Park SJ, Shin MJ, et al. Lobular capillary hemangioma of the hypopharynx. Korean J Otorhinolaryngol Head Neck Surg 2022;65(1):41–44. DOI: 10.3342/kjorl-hns.2020.01081
3. Harris MN, Desai R, Chuang TY, et al. Lobular capillary hemangiomas: an epidemiologic report, with emphasis on cutaneous lesions. J Am Acad Dermatol 2000;42(6):1012–1016. DOI: 10.1067/mjd.2000.104520
4. Amy FT, Enrique DG. Lobular capillary hemangioma in the posterior trachea: a rare cause of hemoptysis. Case Rep Pulmonol 2012;2012:592524. DOI: 10.1155/2012/592524
5. Krstevska V. Early stage squamous cell carcinoma of the pyriform sinus: a review of treatment options. Indian J Cancer 2012;49(2):236–244. DOI: 10.4103/0019-509X.102920
6. Prakash S, Bihari S, Wiersema U. A rare case of rapidly enlarging tracheal lobular capillary hemangioma presenting as difficult to ventilate acute asthma during pregnancy. BMC Pulm Med 2014;14:41. DOI: 10.1186/1471-2466-14-41
7. Nichols GE, Gaffey MJ, Mills SE, et al. Lobular capillary hemangioma. An immunohistochemical study including steroid hormone receptor status. Am J Clin Pathol 1992;97(6):770–775. DOI: 10.1093/ajcp/97.6.770
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